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PubMed1997

[Inclusion body myositis: clinical and myopathological features].

Inclusion body myositis has been recognized as a major form of idiopathic inflammatory myopathy. An old male patient with insidious onset and slowly progressive muscular weakness and artrophy has been reported in this article. The duration of symptom before biopsy was 23 years. The first symptom was dysphagia, and muscular weakness developed seven years later. Muscular atrophy was predominant symmetrically and proximally, particularly the quadriceps femoris muscles. Cervical and abdominal muscles were also affected. Myalgia was absent. Electromyogrophy showed myopathic alterations. Erythrocyte sedimentation rate, creatine kinase, immunoglobulins G increased slightly or moderately. Rheumatoid factor was positive, and he had been diagnosed as having rheumatoid arthritis for 23 years. Inclusion body myositis was ultimately diagnosed based on the muscle biopsy which showed mononuclear cell invasion of nonnecrotic muscle fibers, the characteristic rimmed vacuoles in cryostat sections and cytoplasmic inclusion bodies consisted of plenty of tubulofilaments by electron microscope.

Aged↗
PubMed1999

[A study of clinical symptoms, muscle pathology, therapeutic response, and prognosis of myositis in patients with HTLV-1 associated myelopathy (HAM)].

Myositis is one of well-known complications of HTLV-1 associated myelopathy (HAM). On twelve HAM patients, 10 women and 2 men complicated with myositis, we examined their clinical symptoms, muscle pathology, and therapeutic response. Clinical examination revealed gait disturbance in 10, weakness of upper limbs in 6, muscle atrophy in 4, and myalgia in 3 patients. Blood test disclosed elevated serum CK level in 6 patients. Muscle biopsy showed marked inflammatory changes with many necrotic and degenerating fibers in 7 patients, and the rest showed only focal invasion of inflammatory cells. Eight patients received corticosteroid or immunosuppressant therapy. The neurological symptoms were improved in 5 of 8 patients 3 months after the treatment. And only one out of 5 patients whom we followed up 3 years after the treatment showed improvement.

Adult↗
PubMed1999

Dermatomyositis. Diagnosis and evaluation of dermatomyositis, polymyositis, and inclusion-body myositis.

In diseases of an unknown etiology, such as the idiopathic inflammatory myopathies, we must tackle first of all the question of classification and the degree of disease activity before we can institute treatment. The majority of idiopathic inflammatory myopathies are diagnosed clinically and confirmed by biopsy. The presently applicable methods of diagnosis and evaluation of idiopathic inflammatory myopathy have certain limitations, and hence it is necessary to apply new methods of rating the disease activities. Magnetic resonance imaging (MRI) and 99m-technetium muscle-scanning are the latest noninvasive methods for evaluation of disease activities with myositis. Future laboratory methods to determine the numerous myositis-specific autoantibodies will probably enable identification of subsets of these diseases.

Autoantibodies↗
PubMed2000

Inclusion body myositis, muscle blood vessel and cardiac amyloidosis, and transthyretin Val122Ile allele.

Typical of sporadic inclusion body myositis muscle biopsies are vacuolated muscle fibers containing intracellular amyloid deposits and accumulations of "Alzheimer-characteristic" proteins. There is no muscle blood vessel or cardiac amyloidosis. We report on a 70-year-old African-American man homozygous for the transthyretin Val122Ile allele who has both sporadic inclusion body myositis and cardiac amyloidosis. His unique pathological features included transthyretin immunoreactivity in prominent muscle blood vessel amyloid and congophilic amyloid deposits within vacuolated muscle fibers.

Aged↗
PubMedDate not supplied

Inclusion body myositis (IBM).

Clinical, histological, immunohistochemical and ultrastructural features of 5 cases of inclusion body myositis -4 sporadic (s-IBM) and one hereditary (h-IBM) form are described. These patients (3 men, 2 women) had chronic progressive weakness of varying severity in all 4 extremities with sparing of cranial muscles. Elevation of CPK was noted in 2 patients. Electromyography revealed features of myopathy in 4 and additional neurogenic changes in 2 subjects. Clinical diagnosis was often other than inclusion body myositis. Presence of characteristic eosinophilic inclusions within the vacuoles established the diagnosis. The inclusions were congophilic and showed positivity to ubiquitin, beta-amyloid and SMI-31 in the sporadic cases while congophila was absent in the hereditary form. Immunostaining to hyperphosphorylated-tau was negative in both s-IBM and h-IBM. Membraneous whorls were observed at ultrastructural level. None of the patients improved with steroids and trial with other immunosuppressants was unsuccessful.

Adult↗
PubMed2000

Clinical, pathological and magnetic resonance imaging features of focal myositis: report of three cases.

Focal myositis is a rare idiopathic form of inflammatory myositis involving a single muscle. The presentation is often as a soft tissue pseudotumour. Magnetic resonance imaging (MRI) is a useful noninvasive imaging modality to demonstrate focal nature of the lesion. We describe three patients presenting as pseudotumour of the lower leg. MRI showed hyperintense signals in the involved muscle.

Adolescent↗
PubMed2000

Myositis specific antibodies: frequency in different populations.

Myositis specific autoantibodies (MSA) are the most specific markers of idiopathic inflammatory myopathies (IIM). There is no evidence of presence MSA in patients with other neuromuscular or connective tissue diseases. We compared the frequency of MSA in two groups of IIM patients, one from Poland and one from North America and found no significant statistical differences (21% and 25% respectively). There was a significant difference between the occurrence of immunological marker PM-Sci in scleromyositis patients (22.85% in group I and 7.1% in group II). This figure was also greater than those previously reported in North Americans (2-10%) and Japanese (extremely seldom). These findings confirm the association between MSA and several homogenous clinical syndromes: antisynthetases with the antisynthetase syndrome, anti-SRP with severe, resistant to treatment myositis, anti-Mi-2 with classic, benign dermatomyositis. They underscore the importance of including MSA in the routine diagnostic workup of IIM.

Adolescent↗
PubMed2001

[Idiopathic orbital myositis].

Orbital myositis is a rare disorder considered as a subgroup of inflammatory orbital pseudotumors. The pathophysiology is still unknown. Patients typically present with orbital pain exacerbated by eye movement and diplopia. Response to steroids is dramatic. We report a case of idiopathic myositis of the right inferior muscle, which recovered after steroid therapy.

Adult↗
PubMed2001

Periarticular calcinosis associated with anti-Jo-1 antibodies sine myositis. Expanding the clinical spectrum of the antisynthetase syndrome.

We describe a 58-year-old woman who developed interstitial lung disease (ILD), polyarthritis, and anti-Jo-1 antibodies, with no clinical evidence of myositis. Despite successful treatment with corticosteroid and azathioprine for her arthritis and pulmonary condition, she developed deforming arthropathy of the hands, with periarticular calcinosis. The association of anti-Jo-1 antibodies, ILD, and periarticular calcinosis with subluxing arthropathy sine myositis is rare, with few cases reported. This report expands the clinical spectrum of the antisynthetase syndrome, which is broader than previously reported.

Adult↗
PubMedDate not supplied

Pseudomalignant myositis ossificans in children. Description of a case and review of the literature.

The authors report a rare case of pseudomalignant myositis ossificans occurring during childhood. A female aged 10 years with no previous history of trauma came to their observation with findings of pain and progressive swelling in the gluteal region. The severity of the clinical findings and the absence of characteristic ossification in the x-rays obtained at the onset of the disease suggested that neoplastic pathology be excluded. Diagnosis (bone scan, CT scan, MRI, angiography, biopsy) and the progression of the disease, until its complete clinical resolution, are discussed in light of a review of the literature (44 cases of pseudomalignant myositis ossificans during pediatric age). All of the elements that may be of help in diagnosis and capable of avoiding surgical procedures that are either untimely or improper are emphasized.

Child↗
PubMed2002

Subclinical myositis is common in primary Sjögren's syndrome and is not related to muscle pain.

OBJECTIVE: Although muscle pain is common in primary Sjögren's syndrome (SS), the underlying mechanisms are mainly unknown. We studied all patients with SS at our rheumatology unit with respect to muscle pain in general and to fibromyalgia (FM), and correlated clinical data to muscle biopsy findings. METHODS: We investigated 48 patients with SS according to the modified European diagnostic criteria. The ACR criteria for FM were used to subgroup the patients. Muscle biopsy was performed in 36 patients. Light microscope morphology and immunohistochemical expression of MHC class I, MHC class II, and membrane attack complex (MAC) were studied. RESULTS: We found 44% of patients complained of muscle pain; 27% fulfilled the ACR criteria for FM, whereas 17% had other forms of myalgia. Muscle pain could not be related to histopathological findings. Signs of inflammation were found in 26 of 36 biopsies (72%), and inflammation combined with degeneration/regeneration (i.e., histological signs of polymyositis) in 17 biopsies (47%). However, only 5 patients (14%) had clinical as well as histological signs of polymyositis. Eight muscle biopsies (22%) showed histological features of inclusion body myositis (IBM). However, no patient had clinical symptoms suggestive of this disease. Abnormal expression of MHC class I, MHC class II, and MAC was found in 18 (50%), 16 (44%), and 27 (75%) patients, respectively. CONCLUSION: Muscle pain, especially FM, is common in SS. Histopathological signs of myositis are very common in SS. However, muscle symptoms are not related to histological signs of muscle inflammation. IBM-like findings may represent vacuolar myopathic degeneration due to previous subclinical muscle inflammation rather than a specific clinical entity.

Adult↗
PubMed2002

[Inclusion body myositis. Report of 4 cases].

Inclusion body myositis (IBM) is a primary inflammatory myopathy characterized by an older age at presentation. We describe four IBM cases fulfilling Mendell's diagnostic criteria. All patients were older than 60 years at diagnosis and the mean length of time from onset to diagnosis was 5.7 years. Two of them complained of leg weakness with unsteady gait and the other two, of upper limb weakness. Three patients had dysphagia, one of them had diaphragmatic paralysis and another had bilateral blepharoptosis. Histological sections of the muscle biopsy showed mononuclear cell invasion of nonnecrotic muscle fibers, rimmed vacuoles, intracellular amyloid deposits and 16-21 nm tubulofilaments by electron microscopy. Mitochondrial anomalies were found in two cases. Only one patient had transient response to steroid therapy. Our serie shows that clinical presentation of inclusion body myositis includes a broader spectrum than the classical description.

Aged↗
PubMed2001

Effect of hyperbaric oxygen and penicillin in a murine model of streptococcal myositis.

We investigated the effect of hyperbaric oxygen (HBO2) and penicillin therapy in a murine model of group A streptococcal myositis. The thighs of mice were inoculated with Streptococcus pyogenes. Four groups were evaluated: 1) control (n = 13), 2) HBO2 treatment (n = 15), 3) penicillin treatment (n = 12), and 4) penicillin and HBO2 treatment (n = 13). Histologic methods were utilized to prove the existence of myositis and histologic changes in tissues following experimental intramuscular inoculation of mice with Streptococcus pyogenes. Mortality (day of death) and the number of colony forming units (cfu) were measured. Microscopic sections of the left thighs revealed extensive necrosis of muscle with acute inflammatory infiltrate in all groups. Penicillin significantly lowered cfu count in comparison to the control (P < 0.01). Cfu's in group 4 were significantly lower than in group 3 (P < 0.01). Survival was significantly longer in the penicillin group compared to the control (P < 0.01). Survival in the combined treatment group was significantly longer than penicillin alone (P < 0.01). These results suggest that 1) HBO2 treatment alone does not decrease mortality significantly in vivo, 2) penicillin therapy alone improves outcome significantly, and 3) the combined treatment of penicillin and HBO2 exerts synergistic effects in both decreasing bacterial counts in vivo and increasing survival in this model.

Animals↗
PubMed2002

[Deep necrotizing soft tissue infections. Necrotizing fasciitis and necrotizing myositis].

The authors present a review on deep soft tissue infections--necrotizing fasciitis and necrotizing myositis, incl. the classification of myositis into sub-groups according to the causal agent. The authors describe the incidence of these diseases, basic clinical symptoms and therapeutic principles. Therapy is based on surgical treatment, antimicrobial treatment and in indicated cases on hyperbaric oxygen therapy (HBO).

Fasciitis, Necrotizing↗
PubMed2002

Clinical spectrum of inflammatory myositis in South India--a ten year study.

OBJECTIVE: To describe the clinical spectrum of inflammatory myopathies at a referral hospital in South India. METHODS: Patients were assessed for the pattern of muscle involvement, for the presence of arthritis, Raynaud's phenomenon, interstitial lung disease (ILD) and cardiac involvement. Muscle enzymes, electromyogram (EMG) and muscle biopsies were done. RESULTS: Eighty seven patients with inflammatory myopathies were encountered over 10 years. These included 24 with adult polymyositis, 26 with adult dermatomyositis, one with amyopathic dermatomyositis, five with juvenile myositis, one with dermatomysitis following carcinoma breast and 30 with overlap with other connective tissue diseases. There was a female preponderance (M:F = 1:2.35) except in juvenile myosits group (M:F = 1.5:1). The mean age of onset in years was 33.26 in adult polymyositis, 35.03 in adult dermatomyositis, 7.4 in juvenile dermatomyositis, 42 in malignancy-associated dermatomyositis and 25.51 in the overlap group. Proximal muscle weakness was seen in 98.8% patients, dysphagia in 33.3%, distal muscle weakness in 12.5%, respiratory muscle weakness in 9.2% and dysphonia in 4.6%. Other features included arthritis 35.63%, interstitial lung disease (ILD) 9.2%, Raynaud's 5.7%, myocarditis 4.6% and conduction disturbances 1.15%. Eleveated muscle enzymes were seen in 85.1% patients. Eletromyogram was positive in 66.6%. Muscle biopsy was positive in 85.29%. Anti-nuclear antibody was positive in 67.24%. All received steroids, non-responders needed methotrexate (13 patients) or azathioprine (11 patients). Death occurred in 10 (seven with dermatomyositis predominantly due to respiratory involvement and three with overlap). CONCLUSION: There was female preponderance except in juvenile myositis group. Proximal muscle weakness was the commonest feature. ILD was the commonest respiratory problem, while myocarditis was the commonest cardiac problem seen. Response to therapy and prognosis in polymyositis were good with no mortality during the study period. Death in the dermatomyositis group was mainly due to respiratory involvement.

Adolescent↗
PubMed2003

[Radiological spectrum of myositis ossificans circumscripta evolution].

Myositis ossificans circumscripta is a benign pathology of soft tissue occurring in young patients often after localized trauma. Histological and radiological appearances may mimic a malignant neoplasm, mainly sarcoma. We report a case characteristic of myositis ossificans circumscripta, to illustrate diagnostic arguments which are based on their appearance on conventional radiographs, computed tomography and magnetic resonance imaging and on their changes with time.

Adult↗
PubMed2003

Myasthenia gravis and masticatory muscle myositis in a dog.

A 21-month-old, castrated male Vizsla was presented for pelvic limb weakness, difficulty opening his mouth, ptyalism, voice change, and urinary incontinence. Myasthenia gravis and masticatory myositis were diagnosed. The unusual clinical findings, diagnosis, treatment, and case outcome are described, followed by a brief discussion of myasthenia gravis and masticatory myositis.

Animals↗